Development of CARRA/PReS-endorsed consensus Core and Expanded Datasets in childhood-onset systemic lupus erythematosus for international registry-based research.

OBJECTIVES: Childhood-onset systemic lupus erythematosus (cSLE), representing 15%-20% of individuals with SLE, has been difficult to study globally due to differences between registries. This initiative, supported by Childhood Arthritis Rheumatology Research Alliance (CARRA) and Paediatric Rheumatology European Society (PReS), aims to create Core and Expanded cSLE Datasets to standardise and enhance research worldwide. METHODS: 21 international cSLE experts and 4 patients participated in a Delphi process (questionnaires, 2 topic-specific focus groups and 3 virtual consensus meetings) to create 2 standardised cSLE datasets. The Core cSLE Dataset was designed to include data essential to meaningful clinical research across many settings. The Expanded cSLE Dataset was designed for centres able to consistently collect data to address broader research questions. Final data items for the Core and Expanded datasets were determined by consensus defined as >80% agreement) using an adapted nominal group technique and voting. RESULTS: The resulting Core cSLE Dataset contains 46 items, including demographics, clinical features, laboratory results, medications and significant adverse events. The Expanded cSLE Dataset adds 26 additional items and includes patient-reported outcomes. Consensus was also achieved regarding the frequency and time points for data collection: baseline, quarterly follow-up visits, annually and flare visits. CONCLUSION: Standardised Core and Expanded cSLE Datasets for registry-based international cSLE research were defined through the consensus of global experts and patient/caregiver representatives, endorsed by CARRA and PReS. These datasets incorporate disease-specific and patient-specific features, optimised for diverse settings to facilitate international collaborative research for children and adolescents with SLE worldwide.

Interventions to mitigate EHR and documentation burden in health professions trainees: A scoping review.

BACKGROUND: Health professions trainees (trainees) are unique as they learn a chosen field while working within electronic health records (EHR). Efforts to mitigate EHR burden have been described for the experienced health professional (HP), but less is understood for trainees. EHR or documentation burden (EHR burden) affects trainees, although not all trainees use EHRs, and use may differ for experienced HPs. OBJECTIVES: To develop a model of how interventions to mitigate EHR burden fit within the trainee EHR workflow: the Trainee EHR Burden Model. 1) Examine trainee experiences of interventions aimed at mitigating EHR burden(scoping review). 2) Adapt an existing workflow model by mapping included studies(concept clarification). METHODS: We conducted a 4-database scoping review applying PRISMA-ScR guidance, examining scholarly, peer-reviewed studies that measured trainee experience of interventions to mitigate EHR burden. We conducted a concept clarification categorizing, then mapping studies to workflow model elements. We adapted the model to intervenable points for trainee EHR burden. RESULTS: We identified 11 studies examining interventions to mitigate EHR burden that measured trainee experience. Interventions included: curriculum, training, coaching on the existing EHR for both simulated or live tasks; evaluating scribes' impact; adding devices or technology tailored to rounds; team communication or data presentation at end-of shift handoffs. Interventions had varying effects on EHR burden, most commonly measured through surveys, and less commonly, direct observation. Most studies had limited sample sizes, focused on inpatient settings, and physician trainees. CONCLUSION: Few studies measured trainee perspectives of interventions aiming to mitigate EHR burden. Many studies applied quasi-experimental designs and focused on inpatient settings. The Trainee EHR Burden Model, adapted from an existing workflow model, offers a starting place to situate points of intervention in trainee workflow. Further research is needed to design new interventions targeting stages of HP trainee workflow, in a range of clinical settings.

Improving routine mental health screening for depression and anxiety in a paediatric lupus clinic: a quality improvement initiative for enhanced mental healthcare.

BACKGROUND: Mental health (MH) conditions are prevalent in adolescents with childhood-onset SLE (cSLE). Early identification is crucial in preventing poor patient outcomes; however, MH screening rates remain low. LOCAL PROBLEM: From July 2021-January 2022, only 15% of adolescents in a paediatric tertiary care cSLE clinic were being screened for depression and anxiety. By November 2023, we aimed to increase the percentage of patients with cSLE (≥12-18 years) screened for depression (Patient Health Questionnaire: PHQ-9) and anxiety (Generalised Anxiety Disorder-7: GAD-7) from 15% to 80%. METHODS: This quality improvement project employed the Model for Improvement framework. Stakeholders included the clinic team, patients and families, and MH providers. Statistical process control charts were used to analyse the outcome measure for percentage of screened patients with cSLE. Patient and caregiver satisfaction surveys were conducted at baseline and after screening as a balancing measure. INTERVENTIONS: MH screening workflow with a referral algorithm was developed with stakeholders. Additional interventions included two MH training workshops for healthcare providers and a preclinic reminder of eligible patients for screening. RESULTS: Over 21 months, 146 patients with cSLE completed 270 MH screens, increasing the screening rate from 15%, peaking at 100%, to a median of 56%. Sixty-six individuals (45%) reported symptoms of depression and/or anxiety on their initial screen. Of 270 screens, 44 individuals (17%) reported moderate to severe symptoms meeting the screening workflow criteria for referral to a MH service; 10% of patients screened were referred and seen by the MH service within 2-12 weeks. Patients and caregivers reported satisfaction with the MH screening process and quality of MH follow-up. CONCLUSION: Despite not sustainably meeting the target, MH screening rates increased in the cSLE clinic by nearly fourfold, demonstrating feasibility and acceptability. Patients expressed satisfaction with their mental health follow-up, emphasising its importance in their care.

Toward a responsible future: recommendations for AI-enabled clinical decision support.

BACKGROUND: Integrating artificial intelligence (AI) in healthcare settings has the potential to benefit clinical decision-making. Addressing challenges such as ensuring trustworthiness, mitigating bias, and maintaining safety is paramount. The lack of established methodologies for pre- and post-deployment evaluation of AI tools regarding crucial attributes such as transparency, performance monitoring, and adverse event reporting makes this situation challenging. OBJECTIVES: This paper aims to make practical suggestions for creating methods, rules, and guidelines to ensure that the development, testing, supervision, and use of AI in clinical decision support (CDS) systems are done well and safely for patients. MATERIALS AND METHODS: In May 2023, the Division of Clinical Informatics at Beth Israel Deaconess Medical Center and the American Medical Informatics Association co-sponsored a working group on AI in healthcare. In August 2023, there were 4 webinars on AI topics and a 2-day workshop in September 2023 for consensus-building. The event included over 200 industry stakeholders, including clinicians, software developers, academics, ethicists, attorneys, government policy experts, scientists, and patients. The goal was to identify challenges associated with the trusted use of AI-enabled CDS in medical practice. Key issues were identified, and solutions were proposed through qualitative analysis and a 4-month iterative consensus process. RESULTS: Our work culminated in several key recommendations: (1) building safe and trustworthy systems; (2) developing validation, verification, and certification processes for AI-CDS systems; (3) providing a means of safety monitoring and reporting at the national level; and (4) ensuring that appropriate documentation and end-user training are provided. DISCUSSION: AI-enabled Clinical Decision Support (AI-CDS) systems promise to revolutionize healthcare decision-making, necessitating a comprehensive framework for their development, implementation, and regulation that emphasizes trustworthiness, transparency, and safety. This framework encompasses various aspects including model training, explainability, validation, certification, monitoring, and continuous evaluation, while also addressing challenges such as data privacy, fairness, and the need for regulatory oversight to ensure responsible integration of AI into clinical workflow. CONCLUSIONS: Achieving responsible AI-CDS systems requires a collective effort from many healthcare stakeholders. This involves implementing robust safety, monitoring, and transparency measures while fostering innovation. Future steps include testing and piloting proposed trust mechanisms, such as safety reporting protocols, and establishing best practice guidelines.

Intravenous Golimumab in Children With Polyarticular-Course Juvenile Idiopathic Arthritis: Long-Term Extension of an Open-Label Phase III Study.

OBJECTIVE: To report pharmacokinetics (PK), immunogenicity, clinical effect, and safety of intravenous (IV) golimumab in children with active polyarticular-course juvenile idiopathic arthritis (pcJIA) who participated in A Study to Evaluate the Pharmacokinetics, Efficacy and Safety of Intravenous Golimumab in Pediatric Participants With Active Polyarticular Course Juvenile Idiopathic Arthritis Despite Methotrexate Therapy (GO-VIVA)'s open-label, long-term extension (LTE) through week 252. METHODS: GO-VIVA participants who continued IV golimumab (80 mg/m2 every 8 weeks) after week 52 were included. PK and safety were assessed through week 244 (last dose) and week 252, respectively, and clinical response through week 116. Clinical outcomes included JIA-American College of Rheumatology (ACR) responses and clinical Juvenile Arthritis Disease Activity Score in 10 joints (cJADAS10). Binary outcomes used nonresponder imputation, and other descriptive analyses used observed data. RESULTS: Of 112/127 (88.2%) participants entering the LTE, 69 completed the week 252 visit. Median steady-state trough golimumab concentrations were generally maintained from week 52 through week 244 (range 0.3-0.6 µg/mL). Antigolimumab antibody rates were consistent through week 52 (39.2% [49/125]) and week 244 (44.8% [56/125]). Week 52 JIA-ACR 30/50/70/90 response rates (75.6% [96/127], 74% [94/127], 65.4% [83/127], and 48.8% [62/127], respectively) were generally maintained through week 116 (72.4% [92/127], 71.7% [91/127], 63.8% [81/127], and 50.4% [64/127], respectively), when the median cJADAS10 was 1.6 and 56.7% (72/127) of participants achieved cJADAS10 ≤ 5 (minimal disease activity). Rates (per 100 patient-years) of serious adverse events and serious infections through week 252 were 7.7 and 3.9, respectively. CONCLUSION: GO-VIVA LTE participants experienced adequate PK exposure and stable safety and immunogenicity. The majority of participants experienced no more than minimal residual disease activity. Data suggest IV golimumab treatment provided durable clinical response through week 116, with an acceptable risk-benefit profile.

Defining Documentation Burden (DocBurden) and Excessive DocBurden for All Health Professionals: A Scoping Review.

OBJECTIVES: Efforts to reduce documentation burden (DocBurden) for all health professionals (HP) are aligned with national initiatives to improve clinician wellness and patient safety. Yet DocBurden has not been precisely defined, limiting national conversations and rigorous, reproducible, and meaningful measures. Increasing attention to DocBurden motivated this work to establish a standard definition of DocBurden, with the emergence of excessive DocBurden as a term. METHODS: We conducted a scoping review of DocBurden definitions and descriptions, searching six databases for scholarly, peer-reviewed, and gray literature sources, using the Preferred Reporting Items for Systematic Reviews and Meta-Analyses Extensions for Scoping Review guidance. For the concept clarification phase of work, we used the American Nursing Informatics Association's Six Domains of Burden Framework. RESULTS: A total of 153 articles were included based on a priori criteria. Most articles described a focus on DocBurden, but only 18% (n = 28) provided a definition. We define excessive DocBurden as the stress and unnecessarily heavy work an HP or health care team experiences when usability of documentation systems and documentation activities (i.e., generation, review, analysis, and synthesis of patient data) are not aligned in support of care delivery. A negative connotation was attached to burden without a neutral state in included sources, which does not align with dictionary definitions of burden. CONCLUSION: Existing literature does not distinguish between a baseline or required task load to conduct patient care resulting from usability issues (DocBurden), and the unnecessarily heavy tasks and requirements that contribute to excessive DocBurden. Our definition of excessive DocBurden explicitly acknowledges this distinction, to support development of meaningful measures for understanding and intervening on excessive DocBurden locally, nationally, and internationally.

Sensory-Motor-Oral Stimulation Combined with Early Sucking During the Mandibular Distraction Osteogenesis Process in Children with Robin Sequence.

OBJECTIVE: To describe the findings of children with Robin Sequence (RS) who received sensory-motor-oral stimulation combined with early sucking during mandibular distraction osteogenesis (MDO), compared with children who did not receive the intervention. DESIGN: A quasi-experimental study. Setting: A tertiary public hospital. Patients: Children with RS referred to MDO. A historical group from the same population but managed according to the institution's standard protocol (no sucking) served as a control group. Interventions: Sensory-motor-oral stimulation, including sucking, starting 24 h after MDO (intervention group). Main Outcome Measure: Our hypothesis is that sensory-motor-oral stimulation, including sucking during the DOM process, do not negatively affect surgical outcomes. RESULTS: Twenty-nine children were included. Eight (72.7%) of the 11 patients in the intervention group and 13 (72.2%) of the 18 controls had MDO complications, with no significant difference between the groups (p = 1.000). The most common surgical outcome was antibiotic therapy for surgical site infection (76.2%). Six months after MDO, 22 (75.9%) children attained full oral feeding or associated with alternative feeding methods. CONCLUSION: The intervention group did not have higher complication rates, from a surgical point of view, than control group. The protocol adopted by some centers that contraindicates sucking during MDO should be revised to consider the benefits of such stimulation. Keywords: Pierre Robin Syndrome, deglutition, therapeutics, child development.

Four dimensions of naturalistic language production in aphasia after stroke.

There is a rich tradition of research on the neuroanatomical correlates of spoken language production in aphasia using constrained tasks (e.g., picture naming), which offer controlled insights into the distinct processes that govern speech and language (i.e., lexical-semantic access, morphosyntactic construction, phonological encoding, speech motor programming/execution). Yet these tasks do not necessarily reflect everyday language use. In contrast, naturalistic language production (also referred to as connected speech or discourse) more closely approximates typical processing demands, requiring the dynamic integration of all aspects of speech and language. The brain bases of naturalistic language production remain relatively unknown, however, in part because of the difficulty in deriving features that are salient, quantifiable, and interpretable relative to both speech-language processes and the extant literature. The present cross-sectional observational study seeks to address these challenges by leveraging a validated and comprehensive auditory-perceptual measurement system that yields four explanatory dimensions of performance-Paraphasia (misselection of words and sounds), Logopenia (paucity of words), Agrammatism (grammatical omissions), and Motor speech (impaired speech motor programming/execution). We used this system to characterize naturalistic language production in a large and representative sample of individuals with acute post-stroke aphasia (n = 118). Scores on each of the four dimensions were correlated with lesion metrics, and multivariate associations among the dimensions and brain regions were then explored. Our findings revealed distinct yet overlapping neuroanatomical correlates throughout the left-hemisphere language network. Paraphasia and Logopenia were associated primarily with posterior regions, spanning both dorsal and ventral streams, which are critical for lexical-semantic access and phonological encoding. In contrast, Agrammatism and Motor speech were associated primarily with anterior regions of the dorsal stream that are involved in morphosyntactic construction and speech motor planning/execution respectively. Collectively, we view these results as constituting a brain-behavior model of naturalistic language production in aphasia, aligning with both historical and contemporary accounts of the neurobiology of spoken language production.

Evaluation of depressive and anxiety symptoms in childhood-onset systemic lupus erythematosus: Frequency, course, and associated risk factors.

BACKGROUND: Depressive and anxiety symptoms are common in childhood-onset systemic lupus erythematosus (cSLE), yet their etiology and course remain unclear. We investigated the frequency of depressive and anxiety symptoms longitudinally in youth with cSLE, and associated socio-demographic and disease factors. METHODS: Participants 8-18 years with cSLE completed baseline measures [demographic questionnaire, Center for Epidemiologic Studies Depression Scale for Children (CES-DC), Screen for Childhood Anxiety Related Disorders (SCARED), and psychiatric interview] and follow-up measures (CES-DC and SCARED) > 6 months later. Prevalence of clinically significant depressive (score >15 on CES-DC) or anxiety symptoms (score ≥25 on SCARED) was calculated at baseline and follow-up. Baseline psychiatric interview diagnoses were tabulated. Relationships between socio-demographics (neighborhood-level material deprivation, ethnic concentration, adverse childhood event history, psychiatric condition in a first-degree relative), disease-related factors (disease duration, major organ disease, disease activity, glucocorticoid use, comorbid medical condition) and baseline depressive and anxiety scores, were examined in linear regression models. Factors with univariate associations with p < 0.2 were included in multivariable adjusted models. RESULTS: At baseline, of 51 participants with a mean disease duration of 4.3 years (SD 2.7), 35% (n = 18) and 35% (n = 18) had clinically significant depressive and anxiety symptoms, respectively. Anxiety disorder was diagnosed by psychiatric interview in 14% (n = 7), depressive disorders in 6% (n = 3), and post-traumatic stress disorder in 4% (n = 2). Adverse childhood events and first-degree relative with psychiatric condition were present in 40% (n = 20) and 37% (n = 18), respectively. In multivariable regression analysis, baseline depressive symptoms were positively correlated with neighbourhood-level material deprivation (ß = 4.2, 95% CI [1.0, 7.3], p = 0.01) and psychiatric condition in a first-degree relative (ß = 7.3, 95% CI [2.2, 12.4], p = 0.006). No associations were found between baseline anxiety scores and patient factors. At a median follow-up of 13.5 months (IQR 10.5, 18) for CES-DC (n = 34) and SCARED (n = 44), depressive and anxiety symptoms were persistent (18%, n = 6; 16%, n = 7), and newly present (24%, n = 8; 16% n = 7) at follow-up. CONCLUSION: In this sample, depressive and anxiety symptoms were prevalent and persistent. Depressive symptoms correlated with neighborhood-level material deprivation, and family psychiatric history. These findings support routine psychosocial assessment in cSLE, and provision of appropriate resources.

Clinical outcomes before and after videofluoroscopic swallow study in children 24 months of age or younger.

OBJECTIVE: To evaluate the combined impact of videofluoroscopic swallow study (VFSS) and therapeutic feeding and swallowing interventions on clinical outcomes in children with oropharyngeal dysphagia (OPD). METHODS: This was an uncontrolled longitudinal analytical study in which OPD patients were evaluated before and after VFSS. Children ≤ 24 months of age diagnosed with OPD in a clinical setting and undergoing VFSS for investigation and management of OPD were included in the study. The study participants received therapeutic feeding and swallowing interventions after having undergone VFSS, being followed at an outpatient clinic for pediatric dysphagia in order to monitor feeding and swallowing difficulties. Respiratory and feeding outcomes were compared before and after VFSS. RESULTS: Penetration/aspiration events were observed in 61% of the VFSSs (n = 72), and therapeutic feeding and swallowing interventions were recommended for 97% of the study participants. After the VFSS, there was a reduction in the odds of receiving antibiotic therapy (OR = 0.007) and in the duration of antibiotic therapy (p = 0.014), as well as in the odds of being admitted to hospital (p = 0.024) and in the length of hospital stay (p = 0.025). A combination of oral and enteral feeding became more common than oral or enteral feeding alone (p = 0.002). CONCLUSIONS: A high proportion of participants exhibited penetration/aspiration on VFSS. Therapeutic feeding and swallowing interventions following a VFSS appear to be associated with reduced respiratory morbidity in this population.

Global State of the Art and Science of Childhood Dysphagia: Similarities and Disparities in Burden.

Feeding/swallowing and airway protection are complex functions, essential for survival, and continue to evolve throughout the lifetime. Medical and surgical advances across the globe have improved the long-term survival of medically complex children at the cost of increasing comorbidities, including dysfunctional swallowing (dysphagia). Dysphagia is prominent in children with histories of preterm birth, neurologic and neuromuscular diagnoses, developmental delays, and aerodigestive disorders; and is associated with medical, health, and neurodevelopmental problems; and long-term socioeconomic, caregiver, health system, and social burdens. Despite these survival and population trends, data on global prevalence of childhood dysphagia and associated burdens are limited, and practice variations are common. This article reviews current global population and resource-dependent influences on current trends for children with dysphagia, disparities in the availability and access to specialized multidisciplinary care, and potential impacts on burdens. A patient example will illustrate some questions to be considered and decision-making options in relation to age and development, availability and accessibility to resources, as well as diverse cultures and family values. Precise recognition of feeding/swallowing disorders and follow-up intervention are enhanced by awareness and knowledge of global disparities in resources. Initiatives are needed, which address geographic and economic barriers to providing optimal care to children with dysphagia.

Multivariate lesion symptom mapping for predicting trajectories of recovery from aphasia.

Individuals with post-stroke aphasia tend to recover their language to some extent; however, it remains challenging to reliably predict the nature and extent of recovery that will occur in the long term. The aim of this study was to quantitatively predict language outcomes in the first year of recovery from aphasia across multiple domains of language and at multiple timepoints post-stroke. We recruited 217 patients with aphasia following acute left hemisphere ischaemic or haemorrhagic stroke and evaluated their speech and language function using the Quick Aphasia Battery acutely and then acquired longitudinal follow-up data at up to three timepoints post-stroke: 1 month (n = 102), 3 months (n = 98) and 1 year (n = 74). We used support vector regression to predict language outcomes at each timepoint using acute clinical imaging data, demographic variables and initial aphasia severity as input. We found that ∼60% of the variance in long-term (1 year) aphasia severity could be predicted using these models, with detailed information about lesion location importantly contributing to these predictions. Predictions at the 1- and 3-month timepoints were somewhat less accurate based on lesion location alone, but reached comparable accuracy to predictions at the 1-year timepoint when initial aphasia severity was included in the models. Specific subdomains of language besides overall severity were predicted with varying but often similar degrees of accuracy. Our findings demonstrate the feasibility of using support vector regression models with leave-one-out cross-validation to make personalized predictions about long-term recovery from aphasia and provide a valuable neuroanatomical baseline upon which to build future models incorporating information beyond neuroanatomical and demographic predictors.

Situating word deafness within aphasia recovery: A case report.

Word deafness is a rare neurological disorder often observed following bilateral damage to superior temporal cortex and canonically defined as an auditory modality-specific deficit in word comprehension. The extent to which word deafness is dissociable from aphasia remains unclear given its heterogeneous presentation, and some have consequently posited that word deafness instead represents a stage in recovery from aphasia, where auditory and linguistic processing are affected to varying degrees and improve at differing rates. Here, we report a case of an individual (Mr. C) with bilateral temporal lobe lesions whose presentation evolved from a severe aphasia to an atypical form of word deafness, where auditory linguistic processing was impaired at the sentence level and beyond. We first reconstructed in detail Mr. C's stroke recovery through medical record review and supplemental interviewing. Then, using behavioral testing and multimodal neuroimaging, we documented a predominant auditory linguistic deficit in sentence and narrative comprehension-with markedly reduced behavioral performance and absent brain activation in the language network in the spoken modality exclusively. In contrast, Mr. C displayed near-unimpaired behavioral performance and robust brain activations in the language network for the linguistic processing of words, irrespective of modality. We argue that these findings not only support the view of word deafness as a stage in aphasia recovery but also further instantiate the important role of left superior temporal cortex in auditory linguistic processing.

Adolescent Health Care Needs and Relationship to Disease in Patients With Childhood-Onset Systemic Lupus Erythematosus.

OBJECTIVE: Our objective was to characterize adolescent health and psychosocial issues in patients with childhood-onset systemic lupus erythematosus (cSLE) and evaluate demographic and disease characteristics associated with adolescent health. METHODS: We retrospectively examined adolescents aged 12 to 18 years with cSLE seen at the Hospital for Sick Children meeting the American College of Rheumatology/Systemic Lupus International Collaborating Clinics classification criteria, assessed by adolescent medicine in the cSLE clinic between 2018 and 2020. Adolescent health issues were characterized using the Home, Education/Employment, Activities, Diet/Drugs, Sexuality, Suicide/mood (HEADDSS) framework. Issues were classified as presenting and/or identified; adolescent health burden was tabulated as the number of distinct adolescent issues per patient. Multiple Poisson regression models examined associations between patient and disease characteristics (age, sex, material deprivation, disease activity, disease damage, and high-dose glucocorticoid exposure) and adolescent health issues. RESULTS: A total of 108 (60%) of 181 adolescents with cSLE were seen by adolescent medicine, with a median of 2 (interquartile range [IQR] 1-3) visits and a median of 2 (IQR 1-5) adolescent health issues during the study period. Common issues were mood (presenting in 21% vs identified in 50%), sleep (27% vs 2%), school and education (26% vs 1%), and nonadherence (23% vs 8%). Psychoeducation was provided by adolescent medicine to 54% of patients. High-dose glucocorticoids (risk ratio [RR] 1.82, 95% confidence interval [CI] 1.41-2.35, P < 0.001), material deprivation (RR 1.17, 95% CI 1.04-1.30, P = 0.007), and lower SLE Disease Activity Index scores (RR 0.95, 95% CI 0.92-0.98, P = 0.004) were associated with higher adolescent health burden. CONCLUSION: Adolescents with cSLE experience many adolescent issues, especially low mood. High-dose glucocorticoids and social marginalization are associated with greater adolescent health burden. This study highlights the importance of addressing adolescent health needs as part of routine care.

Clinical outcomes before and after videofluoroscopic swallow study in children 24 months of age or younger / Desfechos clínicos antes e depois da videofluoroscopia da deglutição em crianças com 24 meses de idade ou menos

ABSTRACT Objective: To evaluate the combined impact of videofluoroscopic swallow study (VFSS) and therapeutic feeding and swallowing interventions on clinical outcomes in children with oropharyngeal dysphagia (OPD). Methods: This was an uncontrolled longitudinal analytical study in which OPD patients were evaluated before and after VFSS. Children ≤ 24 months of age diagnosed with OPD in a clinical setting and undergoing VFSS for investigation and management of OPD were included in the study. The study participants received therapeutic feeding and swallowing interventions after having undergone VFSS, being followed at an outpatient clinic for pediatric dysphagia in order to monitor feeding and swallowing difficulties. Respiratory and feeding outcomes were compared before and after VFSS. Results: Penetration/aspiration events were observed in 61% of the VFSSs (n = 72), and therapeutic feeding and swallowing interventions were recommended for 97% of the study participants. After the VFSS, there was a reduction in the odds of receiving antibiotic therapy (OR = 0.007) and in the duration of antibiotic therapy (p = 0.014), as well as in the odds of being admitted to hospital (p = 0.024) and in the length of hospital stay (p = 0.025). A combination of oral and enteral feeding became more common than oral or enteral feeding alone (p = 0.002). Conclusions: A high proportion of participants exhibited penetration/aspiration on VFSS. Therapeutic feeding and swallowing interventions following a VFSS appear to be associated with reduced respiratory morbidity in this population.

RESUMO Objetivo: Avaliar o impacto conjunto da videofluoroscopia da deglutição (VFD) e intervenções terapêuticas de alimentação e deglutição nos desfechos clínicos em crianças com disfagia orofaríngea (DOF). Métodos: Trata-se de um estudo analítico longitudinal não controlado em que pacientes com DOF foram avaliados antes e depois da VFD. Foram incluídas no estudo crianças com idade ≤ 24 meses e diagnóstico clínico de DOF, submetidas à VFD para a investigação e manejo da DOF. Os participantes do estudo receberam intervenções terapêuticas de alimentação e deglutição após terem sido submetidos à VFD, sendo então acompanhados em um ambulatório de disfagia pediátrica para o monitoramento das dificuldades de alimentação e deglutição. Os desfechos respiratórios e alimentares foram comparados antes e depois da VFD. Resultados: Eventos de penetração/aspiração foram observados em 61% das VFD (n = 72), e intervenções terapêuticas de alimentação e deglutição foram recomendadas a 97% dos participantes do estudo. Após a VFD, houve uma redução das chances de receber antibioticoterapia (OR = 0,007) e da duração da antibioticoterapia (p = 0,014), bem como das chances de internação hospitalar (p = 0,024) e do tempo de internação (p = 0,025). A alimentação por via oral e enteral em conjunto tornou-se mais comum do que a alimentação exclusivamente por via oral ou enteral (p = 0,002). Conclusões: Houve alta proporção de crianças que apresentaram penetração/aspiração na VFD. As intervenções terapêuticas de alimentação e deglutição após a VFD parecem estar associadas à redução da morbidade respiratória nessa população.

Lengthening sleep reduces pain in childhood arthritis: a crossover randomised controlled trial.

OBJECTIVES: Juvenile idiopathic arthritis (JIA) is a common chronic childhood disease and chronic pain is a debilitating feature. A strong link has been shown between poor sleep and pain in JIA. However, the causal direction is unknown. This study's aim was to determine if, in adolescents with JIA, a recommended healthful sleep duration leads to reductions in pain when compared with the restricted sleep (RS) duration that is commonly seen. METHODS: Patients with JIA (12-18 years old; pain score of ≥1 on a visual analogue scale) participated in a randomised, crossover sleep manipulation protocol. The 3-week protocol comprised a baseline week (BL), a week with healthy sleep duration (HSD; 9.5 hours in bed/night) and a RS week (RS; 6.5 hours in bed/night). After BL, participants were randomly assigned to either HSD or RS, and then crossed over to the other condition. Pain was self-assessed using the iCanCope with Pain app. We used Bayesian hierarchical models to estimate the effect of sleep duration on pain. RESULTS: Participants (n=31; mean age=15.0±1.8 years) averaged 1.4 (95% credible interval (CrI) 1.2-1.6) more hours of sleep per night during HSD relative to RS. Compared with RS, HSD resulted in a favourable effect on pain scores (OR 0.61, 95% CrI 0.39-0.95). CONCLUSION: It is possible to have adolescents with childhood arthritis get a healthier sleep duration, and this longer sleep results in reduced pain. These findings complement prior correlational studies and confirm a causal relationship between reduced sleep duration and increased pain. TRIAL REGISTRATION NUMBER: NCT04133662.